Aims. The aim of this study was to produce clinical consensus recommendations about the non-surgical treatment of children with Perthes’ disease. The recommendations are intended to support clinical practice in a condition for which there is no robust evidence to guide optimal care. Methods. A two-round, modified Delphi study was conducted online. An advisory group of children’s orthopaedic specialists consisting of physiotherapists, surgeons, and clinical nurse specialists designed a survey. In the first round, participants also had the opportunity to suggest new statements. The survey included statements related to ‘Exercises’, ‘Physical activity’, ‘Education/information sharing’, ‘Input from other services’, and ‘Monitoring assessments’. The survey was shared with clinicians who regularly treat children with Perthes’ disease in the UK using clinically relevant specialist groups and social media. A predetermined threshold of ≥ 75% for consensus was used for recommendation, with a threshold of between 70% and 75% being considered as ‘points to consider’. Results. A total of 40 participants took part in the first round, of whom 31 completed the second round. A total of 87 statements were generated by the advisory group and included in the first round, at the end of which 31 achieved consensus and were removed from the survey, and an additional four statements were generated. A total of 60 statements were included in the second round and 45 achieved the threshold for consensus from both rounds, with three achieving the threshold for ‘points to consider’. The recommendations predominantly included self-management, particularly relating to advice about exercise and education for children with Perthes’ disease and their families. Conclusion. Children’s orthopaedic specialists have reached consensus on recommendations for non-surgical treatment in Perthes’ disease. These statements will support decisions made in clinical practice and act as a foundation to support clinicians in the absence of robust evidence. The dissemination of these findings and the best way of delivering this care needs careful consideration, which we will continue to explore. Cite this article: Bone Joint J 2024;106-B(5):501–507

Aims. There is a lack of high-quality research investigating outcomes of Ponseti-treated idiopathic clubfeet and correlation with relapse. This study assessed clinical and quality of life (QoL) outcomes using a standardized core outcome set (COS), comparing children with and without relapse. Methods. A total of 11 international centres participated in this institutional review board-approved observational study. Data including demographics, information regarding presentation, treatment, and details of subsequent relapse and management were collected between 1 June 2022 and 30 June 2023 from consecutive clinic patients who had a minimum five-year follow-up. The clubfoot COS incorporating 31 parameters was used. A regression model assessed relationships between baseline variables and outcomes (clinical/QoL). Results. Overall, 293 patients (432 feet) with a median age of 89 months (interquartile range 72 to 113) were included. The relapse rate was 37%, with repeated relapse in 14%. Treatment considered a standard part of the Ponseti journey (recasting, repeat tenotomy, and tibialis anterior tendon transfer) was performed in 35% of cases, with soft-tissue release and osteotomies in 5% and 2% of cases, respectively. Predictors of relapse included duration of follow-up, higher initial Pirani score, and poor Evertor muscle activity. Relapse was associated with poorer outcomes. Conclusion. This is the first multicentre study using a standardized COS following clubfoot treatment. It distinguishes patients with and without relapse in terms of clinical outcomes and QoL, with poorer outcomes in the relapse group. This tool allows comparison of treatment methods and outcomes, facilitates information sharing, and sets family expectations. Predictors of relapse encourage us to create appropriate treatment pathways to reduce relapse and improve outcome. Cite this article: Bone Joint J 2024;106-B(7):735–743

Aims. The diagnosis of developmental dysplasia of the hip (DDH) is challenging owing to extensive variation in paediatric pelvic anatomy. Artificial intelligence (AI) may represent an effective diagnostic tool for DDH. Here, we aimed to develop an anteroposterior pelvic radiograph deep learning system for diagnosing DDH in children and analyze the feasibility of its application. Methods. In total, 10,219 anteroposterior pelvic radiographs were retrospectively collected from April 2014 to December 2018. Clinicians labelled each radiograph using a uniform standard method. Radiographs were grouped according to age and into ‘dislocation’ (dislocation and subluxation) and ‘non-dislocation’ (normal cases and those with dysplasia of the acetabulum) groups based on clinical diagnosis. The deep learning system was trained and optimized using 9,081 radiographs; 1,138 test radiographs were then used to compare the diagnoses made by deep learning system and clinicians. The accuracy of the deep learning system was determined using a receiver operating characteristic curve, and the consistency of acetabular index measurements was evaluated using Bland-Altman plots. Results. In all, 1,138 patients (242 males; 896 females; mean age 1.5 years (SD 1.79; 0 to 10) were included in this study. The area under the receiver operating characteristic curve, sensitivity, and specificity of the deep learning system for diagnosing hip dislocation were 0.975, 276/289 (95.5%), and 1,978/1,987 (99.5%), respectively. Compared with clinical diagnoses, the Bland-Altman 95% limits of agreement for acetabular index, as determined by the deep learning system from the radiographs of non-dislocated and dislocated hips, were -3.27° - 2.94° and -7.36° - 5.36°, respectively (p < 0.001). Conclusion. The deep learning system was highly consistent, more convenient, and more effective for diagnosing DDH compared with clinician-led diagnoses. Deep learning systems should be considered for analysis of anteroposterior pelvic radiographs when diagnosing DDH. The deep learning system will improve the current artificially complicated screening referral process. Cite this article: Bone Joint J 2020;102-B(11):1574–1581

Aims

Given the possible radiation damage and inaccuracy of radiological investigations, particularly in children, ultrasound and superb microvascular imaging (SMI) may offer alternative methods of evaluating new bone formation when limb lengthening is undertaken in paediatric patients. The aim of this study was to assess the use of ultrasound combined with SMI in monitoring new bone formation during limb lengthening in children.

Aims. The health-related quality of life (HRQoL) of paediatric patients with orthopaedic conditions and spinal deformity is important, but existing generic tools have their shortcomings. We aim to evaluate the use of Paediatric Quality of Life Inventory (PedsQL) 4.0 generic core scales in the paediatric population with specific comparisons between those with spinal and limb pathologies, and to explore the feasibility of using PedsQL for studying scoliosis patients’ HRQoL. Methods. Paediatric patients attending a speciality outpatient clinic were recruited through consecutive sampling. Two groups of patients were included: idiopathic scoliosis, and paediatric orthopaedic upper and lower limb condition without scoliosis. Patients were asked to complete PedsQL 4.0 generic core scales, Youth version of 5-level EuroQol-5-dimension questionnaire, and Refined Scoliosis Research Society 22-item (SRS-22r) questionnaire. Statistical analyses included scores comparison between scoliosis and limb pathology patients using independent-samples t-test, and correlation tests of PedsQL and SRS-22r. Results. A total of 566 paediatric patients were recruited: 357 (63.0%) having idiopathic scoliosis, and 209 (37.0%) with limb conditions. Patients with limb pathology had lower functioning scale, summary, and total scores of PedsQL than scoliosis patients (p < 0.05 to p < 0.001). No floor or ceiling effects (< 15%) were detected for PedsQL Psychosocial Health Summary and total scores in both groups. PedsQL was sensitive in differentiating patients with/without problems in their daily lives (p < 0.05 to p < 0.01). PedsQL summary and total scores correlated with SRS-22r total score. Conclusion. PedsQL is an effective HRQoL measure for both paediatric orthopaedic groups with minimal ceiling and floor effects, and is capable of detecting worse HRQoL in patients with limb pathology. The multidimensional PedsQL is sensitive in differentiating among those with daily life problems, especially for scoliosis patients. Cite this article: Bone Joint J 2020;102-B(7):890–898

Over a 15-year prospective period, 201 infants with a clinically unstable hip at neonatal screening were subsequently reviewed in a ‘one stop’ clinic where they were assessed clinically and sonographically. Their mean age was 1.62 weeks (95% confidence interval (CI) 1.35 to 1.89). Clinical neonatal hip screening revealed a sensitivity of 62% (mean, 62.6 95%CI 50.9 to 74.3), specificity of 99.8% (mean, 99.8, 95% CI 99.7 to 99.8) and positive predictive value (PPV) of 24% (mean, 26.2, 95% CI 19.3 to 33.0). Static and dynamic sonography for Graf type IV dysplastic hips had a 15-year sensitivity of 77% (mean, 75.8 95% CI 66.9 to 84.6), specificity of 99.8% (mean, 99.8, 95% CI 99.8 to 99.8) and a PPV of 49% (mean, 55.1, 95% CI 41.6 to 68.5). There were 36 infants with an irreducible dislocation of the hip (0.57 per 1000 live births), including six that failed to resolve with neonatal splintage. Most clinically unstable hips referred to a specialist clinic are female and stabilise spontaneously. Most irreducible dislocations are not identified from this neonatal instability group. There may be a small subgroup of females with instability of the hip which may be at risk of progression to irreducibility despite early treatment in a Pavlik harness. A controlled study is required to assess the value of neonatal clinical screening programmes. Cite this article: Bone Joint J 2015;97-B:265-9

Aims. High-quality clinical research in children’s orthopaedic surgery has lagged behind other surgical subspecialties. This study used a consensus-based approach to identify research priorities for clinical trials in children’s orthopaedics. Methods. A modified Delphi technique was used, which involved an initial scoping survey, a two-round Delphi process and an expert panel formed of members of the British Society of Children’s Orthopaedic Surgery. The survey was conducted amongst orthopaedic surgeons treating children in the United Kingdom and Ireland. Results. A total of 86 clinicians contributed to both rounds of the Delphi process, scoring priorities from one (low priority) to five (high priority). Elective topics were ranked higher than those relating to trauma, with the top ten elective research questions scoring higher than the top question for trauma. Ten elective, and five trauma research priorities were identified, with the three highest ranked questions relating to the treatment of slipped capital femoral epiphysis (mean score 4.6/ 5), Perthes’ disease (4.5) and bone infection (4.5). Conclusion. This consensus-based research agenda will guide surgeons, academics and funders to improve the evidence in children’s orthopaedic surgery and encourage the development of multicentre clinical trials. Cite this article: Bone Joint J 2018;100-B:680–4

We performed CT to investigate how treatment may modify the basic skeletal pathology of congenital club foot. Two homogenous groups of patients treated by one of the authors (EI) or under his supervision were studied. The first included 32 patients with 47 club feet reviewed at a mean age of 25 years and treated by manipulation, application of toe-to-groin plaster casts and an extensive posteromedial release. The second included 32 patients with 49 club feet reviewed at a mean age of 19 years and treated by the Ponseti manipulation technique, application of toe-to-groin plaster casts and a limited posterior release. At follow-up the shape of the subtalar, talonavicular and calcaneocuboid joints was found to be altered in many feet in both groups. This did not appear to be influenced significantly by the type of treatment performed. Correction of the heel varus and the increased declination angle of the neck of the talus was better in the club feet of the second group, whereas reduction of the medial subluxation of the navicular was better in the first. There was a marked increase in the external ankle torsion angle in the first group and a moderate increase of this angle in the second group, in which medial subluxation of the cuboid on the anterior apophysis of the calcaneum was always corrected. Equinus was corrected in both groups but three-dimensional CT reconstruction of the whole foot showed that cavus, supination and adduction deformities were corrected much better in the second group

Achilles tenotomy is a recognised step in the Ponseti technique for the correction of idiopathic congenital talipes equinovarus in most percutaneous cases. Its use has been limited in part by concern that the subsequent natural history of the tendon is unknown. In a study of 11 tendons in eight infants, eight tendons were shown to be clinically intact and ten had ultrasonographic evidence of continuity three weeks after tenotomy. At six weeks after tenotomy all tendons had both clinical and ultrasonographic evidence of continuity

We examined 204 children (137 boys and 67 girls) aged 12 years and under with septic arthritis. Their mean age was 31.1 months (1 to 144; SD 41.6). The most common joints affected were the knees and shoulders. Joints in the upper limb were affected more often in younger children and in the lower limb in those who were older. The mean age for an infection was 12 months in the shoulder and 73 months in the hip. The most common organisms cultured were species of Salmonella.

Between 1978 and 1997 all newborns in the Austrian province of Tyrol were reviewed regarding hip dysplasia and related surgery. This involved a mean of 8257 births per year (7766 to 8858). Two observation periods were determined: 1978 to 1982 (clinical examination alone) and 1993 to 1997 (clinical examination and universal ultrasound screening). A retrospective analysis compared the number and cost of interventions due to hip dysplasia in three patient age groups: A, 0 to < 1.5 years; B, ≥ 1.5 to < 15 years; and C, ≥ 15 to < 35 years.

In group A, there was a decrease in hip reductions from a mean of 25.2 (sd 2.8) to 7.0 (sd 1.4) cases per year. In group B, operative procedures decreased from a mean of 17.8 (sd 3.5) to 2.6 (sd 1.3) per year. There was a 75.9% decrease in the total number of interventions for groups A and B.

An increase of €57 000 in the overall cost per year for the second period (1993 to 1997) was seen, mainly due to the screening programme. However, there was a marked reduction in costs of all surgical and non-surgical treatments for dysplastic hips from €410 000 (1978 to 1982) to €117 000 (1993 to 1997). We believe the small proportional increase in costs of the universal ultrasound screening programme is justifiable as it was associated with a reduction in the number of non-surgical and surgical interventions. We therefore recommend universal hip ultrasound screening for neonates.

Aims. A national screening programme has existed in the UK for the diagnosis of developmental dysplasia of the hip (DDH) since 1969. However, every aspect of screening and treatment remains controversial. Screening programmes throughout the world vary enormously, and in the UK there is significant variation in screening practice and treatment pathways. We report the results of an attempt by the British Society for Children’s Orthopaedic Surgery (BSCOS) to identify a nationwide consensus for the management of DDH in order to unify treatment and suggest an approach for screening. Methods. A Delphi consensus study was performed among the membership of BSCOS. Statements were generated by a steering group regarding aspects of the management of DDH in children aged under three months, namely screening and surveillance (15 questions), the technique of ultrasound scanning (eight questions), the initiation of treatment (19 questions), care during treatment with a splint (ten questions), and on quality, governance, and research (eight questions). A two-round Delphi process was used and a consensus document was produced at the final meeting of the steering group. Results. A total of 60 statements were graded by 128 clinicians in the first round and 132 in the second round. Consensus was reached on 30 out of 60 statements in the first round and an additional 12 in the seond. This was summarized in a consensus statement and distilled into a flowchart to guide clinical practice. Conclusion. We identified agreement in an area of medicine that has a long history of controversy and varied practice. None of the areas of consensus are based on high-quality evidence. This document is thus a framework to guide clinical practice and on which high-quality clinical trials can be developed. Cite this article: Bone Joint J 2023;105-B(2):209–214

Aims. Children with spinal dysraphism can develop various musculoskeletal deformities, necessitating a range of orthopaedic interventions, causing significant morbidity, and making considerable demands on resources. This systematic review aimed to identify what outcome measures have been reported in the literature for children with spinal dysraphism who undergo orthopaedic interventions involving the lower limbs. Methods. A PROSPERO-registered systematic literature review was performed following PRISMA guidelines. All relevant studies published until January 2023 were identified. Individual outcomes and outcome measurement tools were extracted verbatim. The measurement tools were assessed for reliability and validity, and all outcomes were grouped according to the Outcome Measures Recommended for use in Randomized Clinical Trials (OMERACT) filters. Results. From 91 eligible studies, 27 individual outcomes were identified, including those related to clinical assessment (n = 12), mobility (n = 4), adverse events (n = 6), investigations (n = 4), and miscellaneous (n = 1). Ten outcome measurement tools were identified, of which Hoffer’s Functional Ambulation Scale was the most commonly used. Several studies used unvalidated measurement tools originally developed for other conditions, and 26 studies developed new measurement tools. On the OMERACT filter, most outcomes reported pathophysiology and/or the impact on life. There were only six patient- or parent-reported outcomes, and none assessed the quality of life. Conclusion. The outcomes that were reported were heterogenous, lack validation and failed to incorporate patient or family perceptions. Until outcomes can be reported unequivocally, research in this area will remain limited. Our findings should guide the development of a core outcome set, which will allow consistency in the reporting of outcomes for this condition. Cite this article: Bone Joint J 2024;106-B(3):277–285

Aims. The aim of this trial was to assess the cost-effectiveness of a soft bandage and immediate discharge, compared with rigid immobilization, in children aged four to 15 years with a torus fracture of the distal radius. Methods. A within-trial economic evaluation was conducted from the UK NHS and personal social services (PSS) perspective, as well as a broader societal point of view. Health resources and quality of life (the youth version of the EuroQol five-dimension questionnaire (EQ-5D-Y)) data were collected, as part of the Forearm Recovery in Children Evaluation (FORCE) multicentre randomized controlled trial over a six-week period, using trial case report forms and patient-completed questionnaires. Costs and health gains (quality-adjusted life years (QALYs)) were estimated for the two trial treatment groups. Regression was used to estimate the probability of the new treatment being cost-effective at a range of ‘willingness-to-pay’ thresholds, which reflect a range of costs per QALY at which governments are typically prepared to reimburse for treatment. Results. The offer of a soft bandage significantly reduced cost per patient (saving £12.55 (95% confidence interval (CI) -£5.30 to £19.80)) while QALYs were similar (QALY difference between groups: 0.0013 (95% CI -0.0004 to 0.003)). The high probability (95%) that offering a bandage is a cost-effective option was consistent when examining the data in a range of sensitivity analyses. Conclusion. In addition to the known clinical equivalence, this study found that the offer of a bandage reduced cost compared with rigid immobilization among children with a torus fracture of the distal radius. While the cost saving was small for each patient, the high frequency of these injuries indicates a significant saving across the healthcare system. Cite this article: Bone Joint J 2024;106-B(6):623–630

Aims. The aim of this study was to determine the consensus best practice approach for the investigation and management of children (aged 0 to 15 years) in the UK with musculoskeletal infection (including septic arthritis, osteomyelitis, pyomyositis, tenosynovitis, fasciitis, and discitis). This consensus can then be used to ensure consistent, safe care for children in UK hospitals and those elsewhere with similar healthcare systems. Methods. A Delphi approach was used to determine consensus in three core aspects of care: 1) assessment, investigation, and diagnosis; 2) treatment; and 3) service, pathways, and networks. A steering group of paediatric orthopaedic surgeons created statements which were then evaluated through a two-round Delphi survey sent to all members of the British Society for Children’s Orthopaedic Surgery (BSCOS). Statements were only included (‘consensus in’) in the final agreed consensus if at least 75% of respondents scored the statement as critical for inclusion. Statements were discarded (‘consensus out’) if at least 75% of respondents scored them as not important for inclusion. Reporting these results followed the Appraisal Guidelines for Research and Evaluation. Results. A total of 133 children’s orthopaedic surgeons completed the first survey, and 109 the second. Out of 43 proposed statements included in the initial Delphi, 32 reached ‘consensus in’, 0 ‘consensus out’, and 11 ‘no consensus’. These 11 statements were then reworded, amalgamated, or deleted before the second Delphi round of eight statements. All eight were accepted as ‘consensus in’, resulting in a total of 40 approved statements. Conclusion. In the many aspects of medicine where relevant evidence is not available for clinicians to base their practice, a Delphi consensus can provide a strong body of opinion that acts as a benchmark for good quality clinical care. We would recommend clinicians managing children with musculoskeletal infection follow the guidance in the consensus statements in this article, to ensure care in all medical settings is consistent and safe. Cite this article: Bone Joint J 2023;105-B(7):815–820

Aims. Medial humeral epicondyle fractures (MHEFs) are common elbow fractures in children. Open reduction should be performed in patients with MHEF who have entrapped intra-articular fragments as well as displacement. However, following open reduction, transposition of the ulnar nerve is disputed. The aim of this study is to evaluate the need for ulnar nerve exploration and transposition. Methods. This was a retrospective cohort study. The clinical data of patients who underwent surgical treatment of MHEF in our hospital from January 2015 to January 2022 were collected. The patients were allocated to either transposition or non-transposition groups. Data for sex, age, cause of fracture, duration of follow-up, Papavasiliou and Crawford classification, injury-to-surgery time, preoperative ulnar nerve symptoms, intraoperative exploration of ulnar nerve injury, surgical incision length, intraoperative blood loss, postoperative ulnar nerve symptoms, complications, persistent ulnar neuropathy, and elbow joint function were analyzed. Binary logistic regression analysis was used for statistical analysis. Results. A total of 124 patients were followed up, 50 in the ulnar nerve transposition group and 74 in the non-transposition group. There were significant differences in ulnar nerve injury (p = 0.009), incision length (p < 0.001), and blood loss (p = 0.003) between the two groups. Binary logistic regression analysis revealed that preoperative ulnar nerve symptoms (p = 0.012) were risk factors for postoperative ulnar nerve symptoms. In addition, ulnar nerve transposition did not affect the occurrence of postoperative ulnar nerve symptoms (p = 0.468). Conclusion. Ulnar nerve transposition did not improve clinical outcomes. It is recommended that the ulnar nerve should not be transposed when treating MHEF operatively. Cite this article: Bone Joint J 2024;106-B(2):212–218

Aims. Brace treatment is the cornerstone of managing developmental dysplasia of the hip (DDH), yet there is a lack of evidence-based treatment protocols, which results in wide variations in practice. To resolve this, we have developed a comprehensive nonoperative treatment protocol conforming to published consensus principles, with well-defined a priori criteria for inclusion and successful treatment. Methods. This was a single-centre, prospective, longitudinal cohort study of a consecutive series of infants with ultrasound-confirmed DDH who underwent a comprehensive nonoperative brace management protocol in a unified multidisciplinary clinic between January 2012 and December 2016 with five-year follow-up radiographs. The radiological outcomes were acetabular index-lateral edge (AI-L), acetabular index-sourcil (AI-S), centre-edge angle (CEA), acetabular depth ratio (ADR), International Hip Dysplasia Institute (IHDI) grade, and evidence of avascular necrosis (AVN). At five years, each hip was classified as normal (< 1 SD), borderline dysplastic (1 to 2 SDs), or dysplastic (> 2 SDs) based on validated radiological norm-referenced values. Results. Of 993 infants assessed clinically and sonographically, 21% (212 infants, 354 abnormal hips) had DDH and were included. Of these, 95% (202 infants, 335 hips) successfully completed bracing, and 5% (ten infants, 19 hips) failed bracing due to irreducible hip(s). The success rate of bracing for unilateral dislocations was 88% (45/51 infants) and for bilateral dislocations 83% (20/24 infants). The femoral nerve palsy rate was 1% (2/212 infants). At five-year follow-up (mean 63 months (SD 5.9; 49 to 83)) the prevalence of residual dysplasia after successful brace treatment was 1.6% (5/312 hips). All hips were IHDI grade I and none had AVN. Four children (4/186; 2%) subsequently underwent surgery for residual dysplasia. Conclusion. Our comprehensive protocol for nonoperative treatment of infant DDH has shown high rates of success and extremely low rates of residual dysplasia at a mean age of five years. Cite this article: Bone Joint J 2023;105-B(8):935–942

Aims. Paediatric fractures are highly prevalent and are most often treated with plaster. The application and removal of plaster is often an anxiety-inducing experience for children. Decreasing the anxiety level may improve the patients’ satisfaction and the quality of healthcare. Virtual reality (VR) has proven to effectively distract children and reduce their anxiety in other clinical settings, and it seems to have a similar effect during plaster treatment. This study aims to further investigate the effect of VR on the anxiety level of children with fractures who undergo plaster removal or replacement in the plaster room. Methods. A randomized controlled trial was conducted. A total of 255 patients were included, aged five to 17 years, who needed plaster treatment for a fracture of the upper or lower limb. Randomization was stratified for age (five to 11 and 12 to 17 years). The intervention group was distracted with VR goggles and headphones during the plaster treatment, whereas the control group received standard care. As the primary outcome, the post-procedural level of anxiety was measured with the Child Fear Scale (CFS). Secondary outcomes included the children’s anxiety reduction (difference between CFS after and CFS before plaster procedure), numerical rating scale (NRS) pain, NRS satisfaction of the children and accompanying parents/guardians, and the children’s heart rates during the procedure. An independent-samples t-test and Mann-Whitney U test (depending on the data distribution) were used to analyze the data. Results. The post-procedural CFS was significantly lower (p < 0.001) in the intervention group (proportion of children with no anxiety = 78.6%) than in the control group (56.8%). The anxiety reduction, NRS pain and satisfaction scores, and heart rates showed no significant differences between the control group and the intervention group. Subanalyses showed an increased effect of VR on anxiety levels in young patients, females, upper limb fractures, and those who had had previous plaster treatment. Conclusion. VR effectively reduces the anxiety levels of children in the plaster room, especially in young girls. No statistically significant effects were seen regarding pain, heart rate, or satisfaction scores. Cite this article: Bone Joint J 2024;106-B(7):728–734

Aims. The Uppföljningsprogram för cerebral pares (CPUP) Hip Score distinguishes between children with cerebral palsy (CP) at different levels of risk for displacement of the hip. The score was constructed using data from Swedish children with CP, but has not been confirmed in any other population. The aim of this study was to determine the calibration and discriminatory accuracy of this score in children with CP in Scotland. Methods. This was a total population-based study of children registered with the Cerebral Palsy Integrated Pathway Scotland. Displacement of the hip was defined as a migration percentage (MP) of > 40%. Inclusion criteria were children in Gross Motor Function Classification System (GMFCS) levels III to V. The calibration slope was estimated and Kaplan-Meier curves produced for five strata of CPUP scores to compare the observed with the predicted risk of displacement of the hip at five years. For discriminatory accuracy, the time-dependent area under the receiver operating characteristic curve (AUC) was estimated. In order to analyze differences in the performance of the score between cohorts, score weights, and subsequently the AUC, were re-estimated using the variables of the original score: the child’s age at the first examination, GMFCS level, head shaft angle, and MP of the worst hip in a logistic regression with imputation of outcomes for those with incomplete follow-up. Results. The discriminatory accuracy of the score in the new population of 367 children was high (AUC 0.78 (95% confidence interval (CI) 0.71 to 0.86)). The calibration of the score was insufficient (slope 0.48 (95% CI 0.31 to 0.65)), and the absolute risks of displacement of the hip in this population were overestimated. The AUC increased with re-estimated weights (0.85 (95% CI 0.79 to 0.91)). Conclusion. The CPUP Hip Score had a high ability to discriminate between children at different levels of risk for displacement of the hip. The score overestimated the absolute risks of displacement in this population, which may have resulted from differences in the way children were initially registered in the two programmes. The results are promising, but the score weights may need re-estimation before its clinical application in Scotland. Cite this article: Bone Joint J 2022;104-B(5):640–644

Aims. Osteofibrous dysplasia (OFD) is a rare benign lesion predominantly affecting the tibia in children. Its potential link to adamantinoma has influenced management. This international case series reviews the presentation of OFD and management approaches to improve our understanding of OFD. Methods. A retrospective review at three paediatric tertiary centres identified 101 cases of tibial OFD in 99 patients. The clinical records, radiological images, and histology were analyzed. Results. Mean age at presentation was 13.5 years (SD 12.4), and mean follow-up was 5.65 years (SD 5.51). At latest review, 62 lesions (61.4%) were in skeletally mature patients. The most common site of the tibial lesion was the anterior (76 lesions, 75.2%) cortex (63 lesions, 62.4%) of the middle third (52 lesions, 51.5%). Pain, swelling, and fracture were common presentations. Overall, 41 lesions (40.6%) presented with radiological deformity (> 10°): apex anterior in 97.6%. A total of 41 lesions (40.6%) were treated conservatively. Anterior bowing < 10° at presentation was found to be related to successful conservative management of OFD (p = 0.013, multivariable logistic regression). Intralesional excision was performed in 43 lesions (42.6%) and a wide excision of the lesion in 19 (18.8%). A high complication rate and surgical burden was found in those that underwent a wide excision regardless of technique employed. There was progression/recurrence in nine lesions (8.9%) but statistical analysis found no predictive factors. No OFD lesion transformed to adamantinoma. Conclusion. This study confirms OFD to be a benign bone condition with low rates of local progression and without malignant transformation. It is important to distinguish OFD from adamantinoma by a histological diagnosis. Focus should be on angular deformity, monitored with full-length tibial radiographs. Surgery is indicated in symptomatic patients and predicted by the severity of the initial angular deformity. Surgery should focus more on the deformity rather than the lesion. Cite this article: Bone Joint J 2022;104-B(2):302–308